Introduction: Neuromuscular disorders (NMDs) cause wasting and weakening of muscles. People affected by NMDs and their carers can experience a number of adverse psychosocial consequences, which can be exacerbated by the rare nature of many of these disorders. This makes access to medical information, timely diagnoses, supportive care and peer support difficult. Options for receiving peer support are increasing through the development of online support groups (OSGs) for people affected by NMDs. Aims: This thesis examined the role of a new OSG in facilitating online peer support for people affected by NMDs and their carers. This was carried out using two distinct, but inter-related, studies. The first study examined the various ways in which members used the message board facility. The second of these studies accessed members’ personal experiences of using this OSG. Methods: In order to examine how members used this new OSG, the first study involved an inductive thematic analysis of OSG message postings. Message postings from the first five months of this OSG’s existence (n=1,914) were analysed, so as to identify the main thematic content of members’ discussions. The second study accessed participants’ personal accounts of their OSG experiences. Semi-structured interviews were conducted with six OSG members and were analysed using Interpretative Phenomenological Analysis (IPA). This interview element examined the context through which members decided to use the group, factors that contributed to their continued use of the group, and the personal impact of participation. Results: The OSG message postings analysis demonstrated how members created a sense of community spirit by establishing common ground through disclosing personal information, searching for connections with people with similar illness experiences or interests, welcoming new members, and sharing aspirations for the development of a resourceful community. Experiences, emotional reactions and support were shared in relation to: delayed diagnosis; symptom interpretation; illness management and progression; and the sense of isolation incurred when managing a rare disorder. The board was also used to discuss societal and political issues pertaining to living with an NMD and methods of raising awareness of such conditions. The results of the interview study showed the valued connection that the OSG gave participants to similar others, in a friendly, non-pressurised environment. Members especially valued the reassurance of knowing that they were not alone in coping with their often-rare condition. The group provided participants with an understanding audience: a rare experience for a group whose condition is not widely known or understood. The information exchanged on the OSG was appreciated due to its specificity in dealing with NMDs. The board was also considered an important platform for raising awareness of NMD-related issues. The gratifying experience of helping other NMD sufferers was highlighted as a key theme. Participants also felt that the OSG was not without its limitations. Difficulties in relating to other people (because of varying disability levels, different disease progression histories, and different views on politics and other interests) influenced participants’ levels of interaction and the perceived benefits of the group. Less common concerns for the group – but important for some individuals – were privacy concerns in using a publicly accessible group and difficulty navigating through the message postings. Conclusions: This thesis provides a novel, in-depth insight into how people used a new OSG for NMDs, and the personal impact of participating in such a group. Analyses of message posting and interview data highlight the vital psychosocial support provided by the OSG, especially given the rarity of many of these conditions. However, it was found that some obstacles to support are inherent in the OSG itself, tempering assessments of the impact of its use.

Background: Knee pain affects 1 in 4 people over 55 years, and is a leading cause of disability in the elderly (Peat et al, 2001). Whilst the prevalence of knee pain has been examined, the natural history of knee pain and associated risk factors remain unknown (O’Reilly, 1996). Objectives: to determine in a community sample over a 10 year period: [1] the incidence of knee pain; [2] the outcome of knee pain; and [3] risk factors for both incidence and outcome of knee pain. Materials and method: This was a retrospective cohort study. Baseline data were collected between 1996-1999, and the cohort was reviewed during 2007-2008. Knee pain was defined as pain around the knee for most days of at least a month. Participants without knee pain at baseline who developed knee pain during the subsequent 10 years were defined as incident cases. Participants with knee pain at baseline who reported worsening of symptoms, improvement of symptoms, no change in symptoms, or who underwent TKR during the past 10 years were defined as outcome cases. Other measures included: age of onset and time from baseline to the first episode of knee pain. Putative risk factors measured at baseline included age, gender and body mass index (BMI); risk factors assessed at follow-up included knee malalignment and foot angulation. Relative risk (RR) was estimated using odds ratio (OR) or hazard ratio (HR) depending on outcomes. Confounding factors were adjusted using logistic regression or COX regression. Results: 9,429 participants were questioned at baseline (2,868 knee pain positive/6,397 knee pain negative). After 10 years, 5,479 were eligible for follow-up. Of them 3,109 responded and 424 underwent x-rays at both baseline and follow-up. The baseline age of this cohort ranged between 40-83 years, with a mean age of 57 years old; 1,725 (55.5%) were women. The incident rate for knee pain cases during the 10 year follow-up period was 742/2,156 (34.4%); this was similar in men (32%) and women (35%). During the 10 year period 250 (27.4%) of the 914 people with pain at baseline experienced worsening of their symptoms, with 81 (8.9%) requiring total knee joint replacements (TKR). A number of risk factors were explored. Obesity (OR 2.19; 95%CI 1.49, 3.22) and varus malalignment (OR 2.82; 95%CI 1.57, 5.06) significantly associated with incident knee pain, whereas back pain (aOR 1.47; 95%CI 1.02, 2.10) and physical work (aOR 1.88; 95%CI 1.02, 3.50) were related to poor outcome. Conclusions: For people over the age of 40 years old, 1 in 3 will develop significant knee pain in the next 10 years. Of people with knee pain, 1 in 4 will worsen over a 10 year period and 1 in 11 will require surgery. A number of risk factors were identified including both systemic/constitutional and more local biomechanical factors. This could have practical implications for primary and secondary prevention particularly in relation to modifiable risk factors, such as reduction in BMI, occupational protection of the knees and possible adjustment of knee malalignment.

Purpose: To describe the experiences of individuals recently diagnosed with fibromyalgia syndrome (FMS) using Arthur Frank's narrative typologies. Relevance: Fibromyalgia syndrome (FMS) is a musculoskeletal condition of unknown aetiology characterised by chronic widespread pain and poor sleep. There is an absence of studies investigating the illness experience of those recently diagnosed. Participants: A theoretical sampling strategy was used to identify 23 individuals (22 female, 1 male) with a first diagnosis of FMS by a Consultant Rheumatologist. Methods: Qualitative in-depth semi-structured interviews (over a 24 month period) were used to identify the perceptions and experiences of individuals recently diagnosed with FMS. The methodological principles underpinning the study were drawn from pragmatism, critical reflection and feminism (data collection). The design was iterative and emergent. Analysis: Interviews were digitally recorded and transcribed verbatim. Narrative thematic analysis was used to analysis the textual data from the interviews. Results: The experience of being diagnosed and living with FMS is biographically disruptive, threatens Identity and challenges patients' understandings of their bodies and the lives they live. Consistent with Arthur Frank's narrative typologies 3 dominant themes of quest, chaos and restitution were identified within the illness experiences of individuals living with FMS. This thesis however challenges Frank's narrative of quest, and proposes the division of this narrative into sub-categories: active engagement and active dis-engagement. Each narrative preference is not uniform but contains common characteristics as it is told through the unique perceptions and experiences of the individual. Conclusions: It is not possible to homogenise the illness experience of individuals with FMS but their narratives contain unifying plotlines. By allowing individuals the opportunity to recount their stories clinicians might recognise these plotlines and understand how the illness experience of FMS is being interpreted.

Background and aims The advent of volar locking plates designed specifically for fractures of the distal radius has resulted in a major shift away from percutaneous fixation of these injuries. However, comparative studies have not always demonstrated better outcomes than those achieved with less invasive and potentially less expensive established techniques. The present study was a randomized controlled trial comparing the outcome of displaced distal radius fractures when treated with a volar locking plate or closed reduction and percutaneous wire fixation, with supplemental bridging external fixation when required. The primary research objective was to ascertain whether the use of volar locking plates improves functional outcome in the short and medium term. The secondary objective was to determine, through economic evaluation, whether the use of volar locking plates for distal radius fractures is of financial benefit to the health service. Methods A single-centre randomized controlled trial of pragmatic design, conducted in a tertiary care institution, with accompanying economic evaluation. 130 patients with displaced distal radius fractures were randomised to either volar locking plate (n=66) or conventional percutaneous fixation methods (n=64). Outcome assessments were conducted at 6 weeks, 12 weeks and 1 year. The primary outcome measure was the PEM score at one year. Secondary outcomes included the QuickDASH, PRWE, EQ-5D and SF-12 scores, range of motion, grip strength, radiographic and cost parameters. A cost-effectiveness analysis was performed from the perspective of the NHS, and in line with NICE guidance on the methods of technology appraisal. “Bottom up” micro-costing methods were used to calculate costs for each treatment pathway, prospectively collecting information on consumables, inpatient and outpatient resource use, complications and additional procedures up to a year post surgery. Main findings Patients in the volar locking plate group had significantly better PEM, QuickDASH, PRWE scores and range of motion at 6 weeks, with no differences at 12 weeks and 1 year. Grip strength was better for the plate group at all time points. The volar locking plate was better at restoring the radiographic parameters of palmar tilt and radial height. Despite the early functional advantage, patients did not return to work sooner. Quality of life scores were marginally, but not significantly, better for the plate group at early follow-up. Both groups returned to baseline at one year. NHS costs for the plate group were significantly higher. For an additional £713, VLP fixation offered 0.018 additional QALYs in the year post surgery. The incremental cost effectiveness ratio (ICER) for VLP fixation at NHS list price was £40,068. Conclusion The current study showed that use of a volar locking plate resulted in better early post-operative function. However, there was no significant difference at, or after 12 weeks. The volar locking plate achieved better radiographic reduction and measured grip strength, but this did not translate to a difference in function at 12 weeks and 1 year. The earlier recovery of function may be of advantage to some patients. However, in spite of their increasing use and popularity, volar locking plates were cost-ineffective according to NICE threshold criteria.

Background: Gout is the most common inflammatory arthritis worldwide. The hallmarks of initial gout presentation are acute pain, swelling, erythema and tenderness in peripheral joints but eventually unremitting arthritis, joint deformity and tophus deposition may develop with long-standing hyperuricaemia. Patients with gout suffer not only arthritis but also cardiovascular, renal, metabolic and other comorbidities. Since gout is the consequence of chronic hyperuricaemia, urate-lowering treatments (ULT) helps prevent the formation of urate crystals and promote dissolution of existing crystals. Methods: This thesis contained results from a series of observational studies relating to gout. The data sources included the Clinical Practice Research Data-link in the UK and the National Health Insurance Database in Taiwan, both of which are representative of the general population in these two countries. Six different analyses were carried out: (1) epidemiology of gout in the UK (chapter 3); (2) epidemiology of gout in Taiwan (chapter 4); (3) Nature history of gout following diagnosis in the UK (chapter 5); (4) familial aggregation and heritability of gout in Taiwan (chapter 6); (5) risk of comorbidities occurring before gout diagnosis (chapter 7) and (6) Effects of allopurinol on all-cause mortality (chapter 8). Results: This study estimated that the prevalence of gout was 2.49% in the UK in 2012 and 6.24% in Taiwan. Incidence of gout was also higher in Taiwan (3.47 per 1,000 person years in 2010) than in the UK (1.77 per 1,000 person years in 2012). The prevalence and incidence were increasing in the UK in the past decade, however, both of which remained stable in the period 2005-2010 in Taiwan. Compared with the general population, individuals with a family history of gout had a two-fold increased risk of the disease. The relative contributions of heritability, shared and non-shared environmental factors to explain phenotypic variance of gout were 35.1%, 28.1% and 36.8% in men and 17.0%, 18.5% and 64.5% in women, respectively. Patients with gout were already at higher risk of multiple comorbidities at diagnosis, furthermore, gout was associated with higher all-cause mortality. In both countries, the management of gout remains poor. Most gout patients were eligible for ULT at diagnosis or shortly after. Allopurinol, the most commonly prescribed ULT in primary care in the UK, exerted a neutral effect on all-cause mortality, which reassures the safety of the drug in terms of all-cause mortality. Conclusions: Gout is the most common inflammatory arthritis affecting one in 40 people in the UK and one in 16 people in Taiwan. It is influenced by both environmental and genetic factors. Both incidence and prevalence keep rising in the UK whilst they remain comparatively stable in Taiwan. The management in the UK remains poor. Primary care physicians should be encouraged to screen for possible existing comorbidities at diagnosis. Most patients are eligible for ULT at diagnosis or shortly after diagnosis, and given the many benefits of ULT for gout patients, early discussion of ULT with patients seems reasonable practice. Allopurinol is not associated with heightened mortality which should reassure practitioners who avoid ULT for fear of serious adverse events.

Introduction: Placebo has been proven effective in many diseases but whether it is effective in the treatment of fibromyalgia, a chronic widespread pain condition affecting 2% of general population, is unknown. Objectives: [1] to determine whether placebo is effective for fibromyalgia; [2] to identify the possible determinants of the placebo effect [3] to gain knowledge around placebo effect, including nocebo effect and placebo response in difference conditions. Method: Literatures were searched for randomised controlled trials that included placebo as a treatment or comparator in people with fibromyalgia. The placebo effect was measured as the improvement of pain and other outcomes from baseline. The effect was compared with no treatment control group or waiting list group. Meta-analysis was undertaken to combine data from different studies. Subgroup analysis was conducted to identify possible determinants of the placebo effect. Results: 3375 studies were found from the literature search. After scrutiny, 204 trials met the inclusion criteria. Participants who took placebo in the trials had significant improvement in pain, fatigue, sleep quality, physical function, and other main outcomes, while participants in the no treatment controlled group stayed unchanged. The effect size of placebo in pain relief is clinically moderate (ES=0.47, 95%CI 0.37 to 0.56). The effect increased with the strength of the active treatment in the trials, participants’ age and baseline pain severity, but decreased in women and with longer duration of disease. Conclusion: Placebo per se is effective in the treatment of fibromyalgia. The effect varies upon context, suggesting that the treatment effect in fibromyalgia depends on context which may be enhanced with the alternation of non-specific or contextual factors.

Title: The association of physical activity, obesity and injury on the risk of knee osteoarthritis (OA) Purpose: 1) To examine the effect of interactions between physical activity, obesity and injury on the incidence and progression of radiographic and symptomatic knee OA; 2) To establish age and gender specific normative data for knee pain, symptoms, function and knee related quality of life (QOL) as the clinical outcome measures in assessing people with knee OA and to examine their associations with OA risk factors including obesity, injury and physical activity. Methods: 1) Using existing cohort data from Osteoarthritis Initiative (OAI) and Multicenter Osteoarthritis Study (MOST) for interaction analyses Participants without radiographic knee OA at baseline were followed for the incidence of radiographic and symptomatic knee OA. In OAI, the focus was on the tibiofemoral joints (TF) only, so TF-OA was defined as a knee with a Kellgren and Lawrence (KL) grade 2 or greater. In MOST, knee OA was defined as a knee with TF-OA (KL ≥2) and/or patellofemoral- OA (osteophyte ≥2; or joint space narrowing ≥1 plus any cyst, osteophyte, or sclerosis using Osteoarthritis Research Society International atlas). The co-occurrence of radiographic knee OA and the frequent knee symptoms (pain, ache, or stiffness on most days of a month over the past 12 months) at the last follow-up was considered as the incidence of symptomatic knee OA. Progression of radiographic knee OA was determined as either one grade increase in KL score or one grade worsening in joint space narrowing at the last follow-up, in participants with radiographic knee OA at baseline. For the progression of symptomatic knee OA, participants with frequent knee symptoms at baseline were included. An increase of greater than 9.29 points in the total Western Ontario and McMaster Universities Osteoarthritis Index score from baseline to last follow-up was considered as a cut-off point (minimal clinical important worsening) for considering a person with symptom progression. Body mass index (obese/non-obese), injury (yes/no), physical activity (active/inactive), age and gender data were also collected at baseline in both databases. The measures of interactions on both additive and multiplicative scales were computed using the generalized estimation equation. 2) Establishing age and gender specific reference values data for Knee Injury and Osteoarthritis Outcome Score (KOOS) and Oxford Knee Score (OKS) Volunteer participants were recruited via a postal survey. From a list of 25,695 postcodes specified by Nottinghamshire local authorities and in the City of Nottingham, 2,500 postcodes were randomly selected. This was based on the proportion of the population in each local authority and in the City of Nottingham. 2,500 postcodes were then equally and randomly assigned into three age groups of 18-44, 45-69 and ≥70 years old. From each postcode assigned to the specific age group, one name and address was randomly selected. Participants were required to complete the questionnaire booklet once only. The questionnaire booklet consisted of the OKS and the KOOS questionnaires. It also collected information regarding participants’ age, gender, height, weight, history of injury and knee joint replacement and physical activity. Results: Interaction analysis In both cohorts, active and inactive people had a similar risk of incident radiographic or symptomatic knee OA (p > 0.05). This effect was not modified by obesity and/or injury in either cohort (p interactions > 0.05). No significant interactions were also found between physical activity, obesity and injury on the risk of radiographic or symptomatic knee OA progression (p interaction > 0.05). Obese people in both cohorts were significantly at a higher risk of incident radiographic and symptomatic knee OA when compared to non-obese people (p < 0.01); injury also increased the incident risk of knee OA (p < 0.01). There were some evidence of positive interactions between obesity and injury on the risk of incident knee OA. This reached statistical significance on additive and multiplicative scales in OAI (aOR-Symptomatic-multiplicative interaction: 2.83, 95%CI: 1.01 to 7.93; aOR-Symptomatic-additive interaction: 3.13, 95%CI: 0.05 to 6.21) and on additive scale in MOST (aOR- Radiological-additive interaction: 1.51, 95%CI: 0.10 to 2.93). There was no evidence of any statistically significant interaction between obesity and injury on the progressive risk of knee OA. Reference values data The overall response rate was 16.5% (n =414, 45% male, 55% female), with the highest in the middle age group with 24%, 18% in the old age and 8% in young age group. A significant dose response relationship was seen between increasing age and worsening scores of KOOS-Pain; KOOS- Activities of daily living (ADL); KOOS-QOL; and OKS (p < 0.05). The median (M) and inter quartile range (IQ) in old, middle and young age groups were as follows: KOOS-Pain (M, IQ: 91.6, 58.3-100; 94.4, 77.7-100; 100, 80.5-100), KOOS- ADL (M, IQ: 91.1, 59.3-100; 98.5, 77.2-100; 100, 89.7-100), KOOS-QOL (M, IQ: 81.2, 43.7-100; 87.5, 62.5-100; 87.5, 68.7-100), and OKS (M,IQ: 42.3, 29-48; 46, 38-48; 47, 42- 48). The oldest age group had the worst scores in KOOS-Pain, KOOS-ADL; KOOS-QOL; and OKS compared to the young or middle age groups (p < 0.05). However, the differences between young and middle age groups were not statistically significant in any KOOS or OKS scores (p > 0.05). Data were also stratified by gender. There was no gender difference in any KOOS or OKS scores (p > 0.05). Obesity and injury were also found as the strongest predictors for the worsening score in all KOOS and OKS subscale scores (p < 0.05), whereas physical activity was significantly associated with a lower risk of knee related complaints (p < 0.05). Conclusion: Physical activity did not increase the risk of incident or progressive knee OA at any level of obesity and/or injury in middle aged and older people with or at high risk of knee OA. In addition, meeting the minimum physical activity guidelines was significantly associated with lower self-reported knee complaints evaluated by KOOS and OKS. Therefore, moderate levels of physical activity appears to be safe to recommend to the general population and people with or at high risk of knee OA regardless of obesity and injury status. There was also some modest evidence of positive interaction between obesity and injury on the risk of incident knee OA. Hence, weight gain prevention strategies may protect injured people against further increase in the risk of knee OA. This study also provided normative data for KOOS and OKS. The self-reported knee complaints were found to vary with age (not gender) being highest in the oldest age group. This suggests that treatment outcomes in people with knee injury and knee OA should be compared against age-matched reference values from the general population.

Chronic pain is a multifactorial experience combining sensory, affective and cognitive components and the underlying neural activity related to pain is subsequently diverse and encompasses widespread regions of the brain. Functional neuroimaging studies have suggested altered patterns of brain activity relating to chronic pain and in response to acute experimental pain but these findings have been inconsistent. Furthermore, research into chronic knee osteoarthritis pain (one of the most commonly presenting chronic pain states) has to date been lacking. The research here seeks to first assess the consistency of findings within the present literature and subsequently use these findings to motivate investigation into the neural correlates of chronic pain and its impact on brain state using advanced functional magnetic resonance imaging techniques alongside questionnaire-based participant report in a chronic knee osteoarthritis pain cohort. The consistency of previously published functional neuroimaging findings of acute experimental pain in healthy and chronic pain cohorts was first established using coordinate based meta-analysis. A large-scale dataset was collated from the literature including over 200 published articles whilst a new coordinate based meta-analytic algorithm was applied that was shown to improve on the multiple-test correction of those previously available. Pain-related brain activity from healthy controls and chronic pain patient displayed no differences when assessed. Subsequent analysis found that application of a painful stimulus to the clinically affected site in a chronic pain patient displayed significant differences compared to that seen in healthy volunteers in the left lentiform nucleus, middle frontal gyrus and right posterior insula. Investigation into other pain-related brain activity such as pain-anticipatory and intensity related displayed overlapping regional activity within the bilateral anterior insula and anterior cingulate (core regions of the salience network). Pain-related deactivation in healthy controls was significant within the subgenual cingulate and the paracentral lobule. Subsequently, as over 200 studies collated did not prove the sensitivity of evoking experimental pain to elucidate alterations in brain activity related to chronic pain, the next study was motivated to directly study the neural correlates of chronic pain itself. Arterial spin labelling (a functional MRI technique that displays increased sensitivity to tonic stimuli such as chronic pain) was used to study 26 chronic knee osteoarthritis patients and 27 matched healthy controls. Reported pain perception was found to correlate significantly with cerebral blood flow (controlled for age, sex and mean whole-brain cerebral blood flow) in limbic regions in OA patients not consistently seen within acute experimental pain such as the bilateral amygdala and subgenual cingulate. Additionally controlling for reported trait anxiety markedly reduced this relationship but was not found to mediate it when tested. Comparisons between knee osteoarthritis pain patients and matched controls displayed no significant differences. Lastly, as chronic osteoarthritis knee pain patients had been found to show an altered state of pain-related brain activity (as assessed via arterial spin labelling) and that networks such as the salience network had been observed in response to pain anticipation and intensity encoding (as assessed via coordinate based meta-analysis), the final study was motivated to study the effect of chronic knee osteoarthritis pain on the state of the brain. Specifically, using resting-state blood oxygenation level dependent functional MRI that is sensitive to the detection of oscillations and correlations. Via these correlations, brain networks such as the salience, default mode and central executive brain networks are observable at rest. An in depth characterisation of connectivity between- and within-networks was carried out using functional, effective and dynamic connectivity analysis. Functional connectivity was assessed via seed-based correlations and found evidence of altered connectivity (increased anticorrelation) of the salience and default mode networks. Additional altered connectivity was found between the central executive network and the salience network. Effective connectivity analysis (via Granger causality analysis) displayed significantly increased negative outflow also from the salience to the default mode network in line with the functional connectivity finding. Lastly, dynamic functional connectivity analysis (as assessed via a ‘sliding window’ approach in which correlations between brain regions were calculated for subsequent 100-second windows along the functional MRI timeseries) analysis was carried out within each network. Dynamic functional connectivity was found to be significantly reduced within the default mode network of chronic pain patients. The functional differences observed provide evidence to stress the role of negative affect on neural activity underlying perceived chronic knee osteoarthritis pain and the effect of chronic pain on the resting brain. These functional neuroimaging findings have potential implications for understanding the brain in chronic pain and may provide framework for future studies.

Dupuytren’s disease (DD) is a fibroproliferative disorder causing deformity of fingers and disability. Different treatments exist ranging from dividing cords (needle aponeurotomy) through disease excision (fasciectomy) to disease excision with resurfacing with a skin graft (dermofasciectomy). A range of outcome measurements has been used in DD, including angular measurements of finger joints and patient-reported outcome measures (PROMs). This thesis hypothesised that the leading candidate outcome measures are inadequate (subject to bias, invalid, and/or uninterpretable) and that currently there is insufficient evidence to inform patient-centred treatment choice in DD. To investigate this, existing evidence was appraised and studies of validity and interpretability of outcomes were conducted. This comprised a systematic review and meta-analysis of surgical trials, a systematic review of interpretability of outcome measures, cross sectional studies of the validity of leading candidate outcome measures (joint angles, the Disabilities of the Arm, Shoulder and Hand tool (DASH), and the Unité Rhumatologique des Affections de la Main scale (URAM)), and a prospective cohort study of outcome interpretability and variables associated with functional outcome. Key findings: • There were too few trials comparing treatments to inform practice in DD, and methodological quality was generally poor. • There were limited interpretability data to guide the design of future studies. • Dynamism was present in 89% of digits, with mean MCPJ dynamism of 6o and PIPJ dynamism of 14o; 11% of digits exhibited over 30 degrees of dynamism. • Patients had virtually unique goals for surgery, with 26% captured by the URAM • The DASH and the URAM were not structurally valid in factor analysis • The DASH was uninterpretable; the URAM’s minimal important change was 10.5 • The factors associated with poor functional outcome differ from those associated with recurrence. Future work should examine validity for other outcome measures; qualitative investigation of patients’ experiences; and patient-centred high quality randomised controlled trials.

Background: Pregnancy is defined as lasting 40 weeks and birth that occurs before 37 weeks of gestation are classed as preterm, with neonates from as early as 22 weeks gestation surviving, although this is rare. Prematurity is associated with a range of long-term morbidities which negatively influence health and well-being. Infants born at less than 33 weeks gestation are at significantly increased risk for impaired motor development compared with their term peers (Allen, 2008). Deficits in motor skills such as balance, eye-hand co-ordination and manual dexterity have been well documented and are evident in infancy (Wood et al., 2000; Bracewell and Marlow, 2002). Early interventions have demonstrated some success in improving motor skills, but the effective elements were unclear. In addition, interactions between parent and infant can be problematic. Aims: The aim was to develop and evaluate an interactive multimedia parenting intervention, which focuses of motor development of their preterm infant, and to compare parental perceptions of infant capability, parental confidence and parental stress. Methods: The design of the intervention incorporated Social learning theory (Bandura, 1977) as the basis for the parenting intervention. The development of the intervention incorporated findings from a systematic review and a focus group with feedback from relevant healthcare, education professionals and parents of preterm infants. A randomised controlled trial was designed and conducted to evaluate the effectiveness and acceptability of the intervention. In total 168 preterm infants were recruited from five NHS neonatal intensive care units and randomly allocated to an intervention or control arm. The intervention arm consisted of the Helping Our Premature infant ON to better motor skills (HOP-ON) programme and Sharing My Infant’s Learning Experiences (SMILES) programme alongside treatment as usual. The control arm received the SMILES programme alongside treatment as usual. The primary outcome was motor scores on from AIMS and Bayley III developmental assessment. The secondary outcomes were parental perceptions of infant capability, parental confidence and parental stress. Evaluation of the study was conducted following the 12 month assessment. Findings: The primary outcomes of motor scores on the AIMS and Bayley III were not significantly different between the intervention and control groups. Parental perceptions of infant capability were measured with IPIQ and no statistically significant differences were found at 3-4 months corrected age. In relation to parental confidence and stress, these were measured at 3-4 months corrected age and 12 months corrected age using the Parenting Stress Index (PSI) short form. At 12 months corrected age there was a difference between the intervention and control group, with the control group showing significantly higher levels of stress than the intervention group (p < 0.038). Conclusion: The need for interventions with parents and their preterm infants is well established, and although the HOP-ON programme did not significantly improve the motor skills of the preterm infants, the evaluation suggests that parents found the information valuable. HOP-ON is an easy to use intervention, which would benefit from further exploration, along with the reduction on reported stress levels at 12 months.